Journals / Türk Patoloji Dergisi / 2018 / Cilt: 34 - Sayı: 3

Congenital Complete Tracheal Ring in a Neonate: A Case Report

Pages
259–261
Publication Date
2022-11-05
DOI
—
🏛️Bibliographic metadata and full text access for this article are provided via DergiPark.
DergiPark Record ↗
✨

AI Summary & Key Takeaways

AI Insight
⚡ TL;DR(Too Long; Didn't Read)

Here, we report an extreme case of VACTErL/TACrD association presented with congenital complete tracheal ring, encephalocele, bilateral radial agenesis with absent thumbs, equinovalgus deformity on right foot, low-set ears and micrognathia.

📌 3 Key Takeaways

🎯
Objective & Hypothesis:Abnormalities of the upper airway tract lead to congenital high airway obstruction and may complicate neonatal airway management in the delivery room.
📊
Key Findings & Data:Congenital complete tracheal rings are a rare and unusual tracheal anomaly, usually presenting in the neonate or infant as respiratory distress.
💡
Conclusion & Impact:Here, we report an extreme case of VACTErL/TACrD association presented with congenital complete tracheal ring, encephalocele, bilateral radial agenesis with absent thumbs, equinovalgus deformity on right foot, low-set ears and micrognathia.
🔬Design:Research Article
📍Scope:Comprehensive Sample
⚙️Variables:Domain Parameters

Abstract

Abnormalities of the upper airway tract lead to congenital high airway obstruction and may complicate neonatal airway management in the delivery room. Congenital complete tracheal rings are a rare and unusual tracheal anomaly, usually presenting in the neonate or infant as respiratory distress. The clinical presentation can vary from almost asymptomatic patients to near-fatal airway obstruction. It may exist as an isolated entity, or in association with other congenital malformations, in particular, cardiac anomalies along with vascular rings and pulmonary slings. Other associated anomalies have also been reported, for example, chromosomal anomalies, malformation of other parts of the respiratory tract, esophagus and skeletal systems. Here, we report an extreme case of VACTErL/TACrD association presented with congenital complete tracheal ring, encephalocele, bilateral radial agenesis with absent thumbs, equinovalgus deformity on right foot, low-set ears and micrognathia.

🔖

Cite This Article

ÖZDEMİR, S. A., ÖZER, E. A., SÜTÇÜOĞLU, S., CUMURCU, S., İLHAN, Ö., BAYOL, Ü. (2022). Congenital Complete Tracheal Ring in a Neonate: A Case Report. Türk Patoloji Dergisi, Cilt: 34 - Sayı: 3, 259–261. https://ajindex.com/articles/640500

📄 Full Text PDF Viewer

📄

PDF görüntüleyici hazırlanıyor...

References (11)

  1. Valdez Cm, Altmayer SP, Barrow mA, Telles JA, Betat rda S, zen Pr, rosa rF. Encephalocele-radial, cardiac, gastrointestinal, anal/renal anomalies: novel evidence for a new condition? Am J med Genet A. 2014;164A:1085-91. Kallen K, robert E, Castilla EE, mastroiacovo P, Kallen B. relation between oculo-auriculo-vertebral (OAV) dysplasia and three other non-random associations of malformations (VATEr, CHArGE and OEIS). Am J med Genet Part A. 2004;127A:26-34.
  2. Castori m, Brancati F, rinaldi r, Adami L, mingarelli r, Grammatico P, Dallapicolla B. Antenatal presentation of the oculo-auriculo-vertebral spectrum (OAVS). Am J med Genet Part A. 2006;140:1573-9.
  3. Solomon BD, Bear KA, Kimonis V, de Klein A, Scott DA, Shaw-Smith C, Tibboel D, reutter H, Giampietro PF. Clinical geneticists’ views of VACTErL/VATEr association. Am J med Genet Part A. 2012;158A:3087-100.
  4. Loeff DS, Filler rm, Vinograd I, Ein SH, Williams WG, Smith Cr, Bahoric A. Congenital tracheal stenosis: A review of 22 patients from 1965 to 1987. J Pediatr Surg. 1988;23:744-8.
  5. Andrews Tm, Cotton rT, Bailey WW, myer Cm 3rd, Vester Sr. Tracheoplasty for congenital complete tracheal rings. Arch Otolaryngol. 1994;120:1363-9.
  6. Dunham mE, Holinger LD, Becker CL, mavroudis C. management of severe congenital tracheal stenosis. Ann Otol rhinol Laryngol. 1994;103:351-6.
  7. Voland Jr, Benirschke K, Saunders B. Congenital tracheal stenosis with associated cardiopulmonary anomalies. Pediatr Pulmonol. 1986;2:247-49.
  8. Chen JC, Holinger LD. Congenital tracheal anomalies: Pathology study using serial macrosections and review of the literature. Pediatr Pathol. 1994;14:513-37.
  9. Cosentino Cm, Backer CL, Idriss FS, Holinger LD, Gerson Cr, mavroudis C. Pericardial patch tracheoplasty for severe tracheal stenosis in children: Intermediate results. J Pediatr Surg. 1991;26:879-85.
  10. Diaz Em Jr, Adams Jm, Hawkins HK, Smith rJ. Tracheal agenesis. A case report and literature review. Arch Otolaryngol Head Neck Surg. 1989;115:741-5. Wei JL, rodeberg D, Thompson Dm. Tracheal agenesis with anomalies found in both VACTErL and TACrD associations. Int J Pediatr Otorhinolaryngol. 2003;67:1013-7. Solomon BD. VACTErL/VATEr association. Orphanet J rare Dis. 2011;6:56. Kunze J, Huber-Schumacher S, Vogel m. VACTErL plus hydrocephalus: A monogenic lethal condition. Eur J Pediatr. 1992;151:467-8.
  11. Evans JA, Greenberg Cr, Erdile L. Tracheal agenesis revisited: Analysis of associated anomalies. Am J med Genet. 1999;82: 415-22. Evans JA, reggin J, Greenberg